Feasibility, acceptability, and effects of a web-delivered behavioral parent training intervention for rural parents of children with autism spectrum disorder: A protocol

Caregivers of children with autism spectrum disorder (ASD) often report higher levels of stress and mental health issues. Support services and parent training programs may help buffer the effects of caring for a child with ASD. However, due to the national lack of trained ASD providers and disparity of ASD support resources available in rural areas, caregivers often go without support. A possible solution to reach caregivers in rural areas is web-based interventions. This paper describes an ongoing pilot study examining the feasibility, acceptability, and preliminary effects on caregiver well-being and disruptive child behaviors for a web-based parent training program (Attend Behavior) for caregivers of young children (ages 2–11 years old) with autism spectrum disorder (ASD) living in rural areas (trial registration NCT05554198). The intervention is available on the internet as well as a downloadable app for mobile phones. Participants will be invited to use the intervention program for 12-weeks. Prior to using the program, participants will be asked to take a baseline survey assessing depressive symptoms (PROMIS Depression Short Form-6a), caregiver stress (Parenting Stress Index-Short Form), child disruptive behaviors (Home Situations Questionnaire-ASD and Aberrant Behavior Checklist). After 12-weeks, participants will be asked to complete a post-intervention survey with the same measurement scales plus questions regarding intervention acceptability, appropriateness, and feasibility (Acceptability of Intervention, Intervention Appropriateness Measure, and the Feasibility of Intervention Measure). Participants are also invited to partake in a brief 1:1 interview with a study team member to give further feedback regarding the intervention. Study retention and participant app usage data will be examined. Information generated from this pilot study will be used to inform a future larger scale randomized control trial of Attend Behavior.


II. Background and Rationale
3][4] Parents of children with ASD have consistently reported higher levels of stress, depression, anxiety, social isolation, and poorer levels of physical health compared to parents of neurotypical children and parents of children with other various chronic health conditions (e.g.][4][5][6][7][8][9][10][11][12][13] The severity of ASD and disruptive behaviors have been correlated with higher parental stress. 3,14,157][18][19][20] Traditional behavioral treatment approaches such as applied behavioral analysis (ABA), have been shown to benefit children with ASD; 21 however, approaches like ABA do not have high evidence for alleviating parent distress.Treatment approaches are delivered in various formats including parent-mediated delivery seen in parent training (PT) programs. 224][25][26][27] In addition, there is economic benefits of PT interventions for children with behavioral issues as evidence by significant positive returns for investing in PT for youth at risk or already experiencing behavioral difficulties. 28reatment disparities.0][31][32] These disparities include access to services, use of services, and satisfaction and perceived effectiveness of services for children with ASD. 31,33The lack of providers in rural areas have resulted in many rural children with ASD relying on the school systems for interventions despite there being a lack of trained specialists and funding for special education programs in rural schools. 34,35This is problematic as it results in children not receiving early intervention prior to entering the school system and parents being fearful for what will happen once they age out of school. 36 The goal of RUBI is to improve parent response to problem behaviors and prevent disruptive behaviors.26,40 Historically, RUBI has been conducted inperson at a clinic with a provider and parent.Recently, with the COVID-19 pandemic, many providers had to pivot to use technology to provide services from distance.In response to this, Shanok et al. 26 completed a feasibility pilot study using synchronous telehealth services for delivering RUBI to parents of children with ASD.The findings of this study showed that the feasibility of RUBI delivered via telehealth for reducing problem behaviors of children with ASD was consistent with prior RUBI studies. 26While Attend Behavior is used clinically and at times in addition to the RUBI program, there are no current studies published examining the effects of Attend Behavior for parents of children with ASD.There is an overall lack of published studies examining the effects of solely using a mobile-delivered PT program aimed at decreasing disruptive behaviors for children with ASD.One study noted using the application iSTIM to reduce stereotypy behavior. 41Therefore, the proposed study will provide an important contribution to the major gap in knowledge surrounding mobile application delivered ASD PT.

Purpose
Therefore, the purpose of the proposed study is to test the acceptability, feasibility, and preliminary effects of Attend Behavior in an underserved population (rural) of parents of children with ASD.Attend Behavior was chosen for testing based on a rigorous review of the existing parenting programs conducted by the study investigator.Ultimately, Attend Behavior mirrors the evidence-based RUBI parent training program that shows promising results for reducing parental stress and evidence of effectiveness in reducing child behavior problems.Attend Behavior is available on the Web and as a mobile application and can be used independently by parents or in collaboration with a clinician, making it more accessible and versatile.
In this study, we will pilot test Attend Behavior in a sample of 40 parents/guardians with a young child (2-11 years old) with ASD.Parents will provide instrumental feedback on the feasibility and acceptability of Attend Behavior and provide feedback on additional elements that they need in an intervention to support them.Preliminary results of this pilot study will influence future larger randomized control trials.

III. Procedures
A.

Research Design
The proposed pilot study is a single group pre/post-intervention design with both quantitative and qualitative data; therefore, a mixed methods design will be employed.

B. Sample
Parents will be recruited using the following methods: The primary method of recruiting participants in this study is through a community mental health organization, Mid-Ohio Behavioral Health, which has multiple locations in central Ohio including Franklin, Licking, Muskingum, Guernsey, and Coshocton counties.Flyers to promote the study with a QR code and a printed link to direct interested clients to the Redcap survey for assessment of eligibility and collection of contact information will be posted in the Mid-Ohio Behavioral offices for study promotion.The study team will provide education regarding the study including study goals and general information via an email to all staff at Mid-Ohio Behavioral Health.The email will also include the link (to direct interested clients to the Redcap survey for assessment of eligibility and collection of contact information) to provide to their clients if they are interested in the study.

The Learning Spectrum School
A second recruitment site is The Learning Spectrum (TLS) School.TLS is a school focusing on providing education to children with ASD and other developmental disabilities.One of their sites located in Licking county and will be used for study promotion and recruitment.
Flyers to promote the study with a QR code and a printed link to direct interested parents to the Redcap survey for assessment of eligibility and collection of contact information will be posted at TLS-Johnstown for study promotion.The study team will provide education regarding the study including study goals and general information via an email to all staff at TLS-Johnstown.The email will also include the link (to direct interested clients to the Redcap survey for assessment of eligibility and collection of contact information) to provide to their student's parents if they are interested in the study.

Facebook
A third method is Facebook.To recruit via Facebook, the study team will create a Facebook account containing relevant information about the research study's purpose, eligibility, and contact information to learn more.Using the research study's Facebook page, the research team will privately message Facebook group moderators of parents of children with ASD groups asking permission to post information about the study in private pages.With the permission of private group moderators, we will also post the study recruitment flyer in the private groups.This method of recruitment on Facebook has been previously used by the PI in studies as a research assistant and co-investigator and has been exceedingly successful.In addition, an ad on Facebook that was created with collaboration with the Ohio State University CCTS may be published for recruitment.The ad is available in both English and Spanish.Furthermore, rurally located public libraries and primary care offices will be contacted to engage in efforts of recruitment as well.We will send emails to representatives of the above-listed organizations (see template) asking them to pass along the study information and flyer to potentially interested parties.Alternatively, organizations can opt to print the study recruitment flyer and display the flyer in their facility if they so choose.In addition, postcards were created with collaboration with the Ohio States University CCTS and will be offered to be mailed to any organization that reports they would prefer the postcards.

The Ohio State University Extension Offices
Ohio State University (OSU) Extension offices located in rural counties such as Muskingum County, Holmes County, and Knox County will be contacted for aid in recruitment.We will send emails to representatives at the various OSU Extension Offices (see template) asking them to pass along the study information and flyer to potentially interested parties.Alternatively, organizations can opt to print the study recruitment flyer and display the flyer in their facility if they so choose.A request for this project will be created in SFARI to be advertised for potential participants signed up in the SFARI Base to receive information regarding the study.Further information regarding the SFARI Base can be found here: https://www.sfari.org/resource/sfari-base.

Screening procedures
All interested potential participants will be directed by a link and/or QR code on the flyers to a website housing the Redcap survey to screen for eligibility.Participants will answer a short survey to determine eligibility.Only screening data from enrolled participants will be retained.All other screening data from unenrolled or ineligible participants will be destroyed immediately.
If the participant is eligible, they will be prompted to provide their name and contact information (i.e.phone number, preferred time of day for contact, email).The study PI or study team member will contact all eligible interested participants promptly.The verbal consent form will be read in full by the PI the participant over the phone.All consented participants will be emailed a copy of the consent form and contact card.

Sample Size
We will enroll up to 40 parents/guardians into the study.The sample size is deemed sufficient for a pilot study and as we are examining qualitatively feasibility and acceptability of Attend Behavior.

C. Measurement / Instrumentation
Data in this study will be both quantitative and qualitative in nature.Parents will complete selfreported surveys at baseline and post-intervention (T2) using REDCap.For baseline data collection, parents will be emailed or texted a link to the respective survey.There will be two baseline surveys, one for parents of children ages 2-5 years old and another for parents of children ages 6-11 years old.Baseline and T2 surveys will be available in both English and Spanish.For T2, participants will be emailed, texted, and/or messaged on Attend Behavior (participant preference) the link to the survey.The total time commitment for the baseline and T2 surveys is no longer than 30 minutes.Please see below for a description of study assessments: 1.The demographic survey will include both parent and child information that is selfreported by the parent.The demographic survey will be administered at baseline.

2.
The Aberrant Behavior Checklist (ABC).The ABC measures child problem behavior and consists of 58 questions with five subscales: irritability, withdrawal, stereotypic behavior, hyperactivity, and inappropriate speech.The ABC will be administered at baseline and T2.

3.
The Home Situations Questionnaire -Autism Spectrum Disorder (HSQ-ASD).The HSQ-ASD measures child noncompliance in the home and consists of a 24-item rating where parents report yes/no if the child has been noncompliant in the past 4 weeks to a variety of situations in the home setting (e.g."when told to brush teeth").If the child reports the child was noncompliant in a situation within the last 4 weeks, they are prompted to rate the severity of that problem on a 1-9 Likert scale with higher scores indicating higher severity.The HSQ-ASD will be administered at baseline and T2.

4.
The PROMIS Depression -Short Form 6A.This 6-item PROMIS short form measurement prompts participants to rate how often they have felt emotions associated with depression within the past 7 days on a scale of 1 = Never to 5 = Always.The PROMIS will be administered at baseline and T2.

5.
The Parenting Stress Index -Short Form (PSI-SF).The PSI-SF is a 36-item measurement to measure parental stress with three domains: parental distress, parent-child dysfunctional interaction, and difficult child to yield a total stress score.The PSI-SF will be administered at baseline and T2.

IRB Protocol Number:
2021B0434 IRB Approval date: 02/12/2022 Version: 2 6. Acceptability of Intervention (AIM), Intervention Appropriateness Measure (IAM), and the Feasibility of Intervention Measure (FIM).With a total of 12-items answered on a Likert scale of 1 = completely disagree to 5 = completely agree, the AIM, IAM, and FIM is a brief measurement to examine implementation success. 42The AIM, IAM, and FIM will be administered at T2.

7.
The Autism Severity Rating Scale -Short Form (ASRS-SF) (2-5 year old and 6-18 year old).The ASRS-SF (2-5 year old) and the ASRS-SF (6-18 year old) are both 15-item measurement scales to assess the severity of common behaviors associated with Autism Spectrum Disorder.The ASRS-SF will only be given at the baseline survey.If the parent has a child that is 2-5 years old, they will be given the baseline survey that includes the ASRS-SF (2-5 year old) form.If the parent has a child that is 6-11 years old, they will be given the baseline survey that includes the ASRS-SF (6-18 year old) form.

Exit interviews
After the T2 survey is complete, participants will then complete a 20-minute exit interview with the PI.The exit interview will be conducted over the phone or zoom according to participant preference.The purpose of the post-intervention interview is to gauge the preliminary feasibility and acceptability of Attend Behavior.The interview will be guided by a semi-structured interview guide.Interviews will be audio-recorded and transcribed.

D. Detailed study procedures
Once participants have been screened and are eligible, the study PI or study team member will contact the participant for an enrollment meeting.The enrollment meeting will take approximately 15 minutes to complete and take place over the phone.The enrollment meeting will not be audio recorded.The enrollment meeting is comprised of the study team educating the participant on the study, obtaining consent, verifying participant contact information, and providing an overview of the Attend Behavior intervention.The participant will be texted or emailed a link to the baseline survey after the phone call is complete.Once the baseline survey is complete, the PI will email the participant the short video created by Attend Behavior explaining the features of Attend Behavior and how to use the program, a link to the Attend Behavior website, links to download the Attend Behavior mobile application in the Apple Store and the Google Play Store, and their access code to enable their Attend Behavior account.
The enrollment meeting will also include assessing the participant's preference for email, text messages, and/or Attend Behavior messaging reminders to complete the T2 survey and exit interview.Participants will be offered to schedule the exit interview during the enrollment appointment or an option to schedule the interview at a later time.Participants will be reminded that they can Zoom into the 1:1 interview or complete the interview via phone.Participants will be informed that they will receive a survey link after completing the intervention to complete approximately 12-weeks after initiating the intervention.They will be informed that the Attend There will be no hard copies of data in this study.All data (e.g., audio recordings, surveys) will be housed in the Ohio State University College of Nursing's R-drive.Only research personnel (i.e., RA, study investigators) will have access to the R-drive.The R-drive runs Windows Server 2012R2 and is only used to host research files.The Windows operating system is kept up to date by weekly, automatic updates from Microsoft.The physical server is housed in a secure room within Newton Hall.Only pertinent IT staff and the building coordinator have keys to access this room.Electronic access to the research server is restricted to the college LAN or VPN by a Cisco ASA 5510 firewall.Access from the internet without VPN is not allowed.Electronic access is further restricted by the on-host Windows Firewall.User access is tightly controlled, by request only.New users must be cleared by the College of Nursing Associate Dean for Research.Permissions to individual folders and subfolders are managed individually.Users only have access to folders of projects in which they are directly involved.All changes are submitted in writing.Permissions are audited annually by IT in collaboration with the folder/study owner.The administrator account for the server has a non-standard password and has been disabled.All user accounts connecting to the server are domain accounts and are authenticated through a domain controller.Accounts are automatically locked out for 30 minutes after 5 invalid attempts.User access and system information is logged and exported to a central event logging server.All data will be retained for seven years and then destroyed per the policy at Ohio State University.

IRB Protocol Number:
2021B0434 IRB Approval date: 02/12/2022 Version: 2 Data will only be presented in aggregate with no disclosure of individual cases at scientific conferences or in publications.Data will only be transferred to others for analysis when the data is completely de-identified.Five years after the last follow up assessment is completed; any identifying information will be destroyed.Per the American Psychological Association guidelines, the de-identified data will be kept seven years after the final publication of these data.
Discomfort with Questions.We do not anticipate discomfort with questions across study phases as we are asking participants to provide demographic information and to evaluate an intervention.Adverse reactions from answering questions in the assessments are unlikely because these are standard questionnaires used across many populations without complication.Any distress will be minimized by assurances that participants can skip questions they do not feel comfortable answering and withdraw from the study at any time without penalty.
Participants will be encouraged that if at any time during the study they feel distressed, they are encouraged to contact the study team.
Specific Procedures for Abuse, Homicide, and Suicide.
All consent forms will include a statement that if a member of the research team suspects that a child is being abused or neglected, they are required by law to report the concern to child protective services.If the parent has any concern about this, the consent document will encourage the parent to call the toll-free number to discuss those concerns before signing the consent form.Ohio State law mandates that workers in certain professions (e.g., RNs) report if they have reasonable cause to suspect abuse or neglect.Although we do not ask about child abuse or neglect, if during the course of the study something triggers the concern of anyone on the research team related to child abuse or neglect, we will institute procedures for mandated reporting.Public Children's Services Agency (PCSA) of the State of Ohio will be notified by phone and may initiate an investigation.In addition to the placement of the phone call, research staff will send written confirmation to the appropriate child protective services field office within 48 hours.These procedures will satisfy all legal requirements.We do not ask about homicidal or suicidal intent or ideation in this study.However, we have protocols in place should a parent reveal homicidal or suicidal intent or ideation.In the case of homicidal intent, we will report the information to both the police and the intended victim.In the case of suicidal ideation or intent, the PI will assess for a plan, means, and intent.Upon this review, next steps are discussed.For parents who are not deemed acutely suicidal, we will provide a list of referrals for mental health services within the area.If imminent risk is determined in either a parent or child, the participant will be directed to go to the emergency room.If the participant refuses to go to the emergency room and imminent risk of suicide/harm to self is determined, then we will call the police to accompany the participant.If the child's disruptive behavior becomes severe and places the child or others around them at possible harm, the parent will be given a list of resources for higher level of care within the area.

E. Internal Validity
As this is pilot study with a small sample size, there are inherent risks to validity.This study includes a small sample size and therefore, the findings are not generalizability to the broader population of parents with children with ASD.Multiple validated questionnaires are being used to assess outcome data for preliminary effects to reduce threats to internal validity.

Quantitative Data
Quantitative data in this study is derived from the demographic questionnaire, baseline survey, and the post-intervention survey.We will conduct exploratory data analyses to check data accuracy, examine variable distributions, and summarize sample characteristics.Data anomalies once identified will be fully investigated and remedial strategies will be considered as appropriate.Although the full ABC measurement will be included in the baseline and T2 surveys, only the ABC-Irritability subscale (ABC-I) will be used for analyses as the intervention does not target the other symptoms included in the other ABC subscales.For Aim 1, we will use descriptive statistics to examine the acceptability and feasibility of the intervention, including rates of completion of intervention and recruitment and retention rates, as well as average scores of AIM, IAM, and FIM.For Aim 2, descriptive statistics will be used to summarize each outcome (i.e.ABC-I, HSQ-ASD, PROMIS, and PSI-SF).We will report mean differences, 95% confidence intervals, and effect sizes for baseline to post-intervention difference.Mixed-effects models with subject-specific random effects will be further applied to test the intervention effects adjusted for covariates if any.The covariates will be included as fixed-effects in the model and potential interaction effects will be examined.This approach will allow us to account for missing data under the missing at random assumption.

Qualitative data
Qualitative data sources in this study include the exit interview.All audio files will be transcribed verbatim promptly after the interviews.The original audio recordings and transcripts will be stored in the College of Nursing R-drive.A member of the study team will review the transcripts of the audio interviews for accuracy.Prior to data analysis, all qualitative sources will be de-identified with identifiers being redacted.All qualitative data will be analyzed using NVIVO.
NVIVO will assist with the organization and coding of the unstructured narrative data.NVIVO has several capabilities including classifying and arranging data and organizing the data into similar concepts to allows for relationships to emerge.The PI and a member of the research team will read all participant interviews.Codes and coding categories will be compared and discrepancies discussed before establishing a codebook.
Due to the qualitative methodology chosen, data analysis will occur simultaneously with data collection.Data saturation will be attained when no new additional data are revealed in the conceptual categories.The data analysis strategy that will be used in the proposed study is content analysis due to the inductive nature of the work.The aim of content analysis is to provide a thick, rich description of the phenomena of interest using the participant's own words.In qualitative description, the researcher does not infer but rather stays close to the data and will Child with ASD resides at home with the parent c.Parent age is 18 years or older IRB Protocol Number: Reside in a rural (micropolitan or noncore) county e. Has access to either a smartphone with the Apple App store or Google Play store or a computer with Internet access f.Ability to read in English or Spanish Exclusion a.They are currently participating in another behavior PT program or in the past 6-months have been enrolled in another behavior PT program as this could confound the results a.The legal guardian of one or more children ages 2-11 years old with a formal diagnosis of ASD by parent report b.d.